Evidence for telemedicine heterogeneity in rheumatic and musculoskeletal diseases care: a scoping review

Telemedicine and digital health represent alternative approaches for clinical practice; indeed, its potential in healthcare services for prevention, diagnosis, treatment, rehabilitation, and disease monitoring is widely acknowledged. These are all crucial issues to consider when dealing with chronic Rheumatic and Musculoskeletal Diseases (RMDs). The aim was to determine the current state of telemedicine in the field of rheumatology, considering the tools and devices in use as well as the Patient Reported Outcomes. A scoping review was performed following the PRISMA-ScR, retrieving articles through five databases from 1990 to 2022. Inclusion criteria were as follows: (I) adult patients with RMDs, (II) original research papers in the English language with available abstracts, and (III) telehealth and telemedicine are provided as healthcare services. Within the 62 included studies, multiple tools of telemedicine were used: 21/62 websites/online platforms, 18/62 mobile applications, 16/62 telephone contacts, 5/62 video-consultations, and 1/62 wearable devices. Outcomes were classified based on the economic, clinical, and humanistic framework. Clinical outcomes assessed through digital tools were pain, disease activity, and serum uric acid levels. Humanistic outcomes have been grouped according to four categories (e.g., mental and physical function, health management, and health perception). The heterogeneity of digital tools in the field of rheumatology highlights the challenge of implementing reliable research into clinical practice. Effective telerehabilitation models have been presented, and the use of a tight control strategy has also been mentioned. Future research should focus on establishing studies on other RMDs as well as summarizing and formulating clinical guidelines for RMDs. Key Points • Evidence for the usefulness of telemedicine and digital health for managing and monitoring rheumatic and musculoskeletal diseases is progressively increasing. • Several digital tools effectively measure clinical and humanistic and patient reported outcomes in rheumatic and musculoskeletal diseases. • Integrating diverse digital tools in rheumatology is challenging yet promising. • Future research should focus on developing standardized recommendations for practical use of telemedicine in daily practice. Supplementary Information The online version contains supplementary material available at 10.1007/s10067-024-07052-w.


Introduction
About 1.71 billion of the global population is affected by rheumatic musculoskeletal diseases (RMDs), leading to disability, mortality, and morbidity [1,2].Almost one-third of the general population experience a problem linked to RMDs, with a prevalence that ranges from 9.8% to 33.2% [3].The WHO reports that RMDs alone account for half of all chronic diseases, especially in older adults, and are the primary cause of pain and disability in Europe [4].In Italy, 27% out of the 60 million adults more than 5 million experience chronic pain and is diagnosed with a RMD [5].In the last decades, epidemiological data about RMDs has shown an increase due to population aging, higher survival rates, and the spread of early diagnosis.Consequently, an increasing pressure is generated on healthcare services, delaying in some cases the implementation of valid clinical guidelines [6][7][8][9].In the majority of RMDs, chronicity is a key aspect, Extended author information available on the last page of the article requiring intensive patients' monitoring and regular followups, that sometimes can be effectively delivered also through telehealth tools with successful results in terms of user's outcomes (i.e., self-care, engagement, satisfaction) [10][11][12].
The active involvement of patients in the collection of data about their health outcomes through the Patients Reported Outcome Measures (PROMs) is a significant option within busy health services and from the perspective of patient empowerment [3].In addition, periods of disease remission favored by therapeutic innovation/biological therapy allow for patients remote monitoring with specific characteristics of clinical stability (i.e., low disease activity or disease remission) and established diagnosis in some settings, like the outpatient clinics [8,9,12].
The advancement of telemedicine (TM) in rheumatology facilitates the provision of services such as prevention, diagnosis, treatment, rehabilitation, and follow-up, lessening the burden of the disease from the patients and caregivers perspective, yet some aspects still remain to be better defined [11].The term "telehealth" refers to a wider range of non-physician services, including TM, telenursing, telepharmacy, and linguistic interpretation, as well as analog and audio-video out-of-office visits as alternatives to in-person healthcare.These terms can be used interchangeably with integrated remote care modalities, including mobile health and E-health platforms [13].Specifically, the term e-Health has been used since 1990s to refer to the use of information and communication technology to support, improve, or provide access to knowledge and services in healthcare settings [14].However, nowadays, it has come to refer to all services, goods, procedures, and infrastructures related to digitization in the healthcare industry [15].
Tele-rheumatology originated prior to the COVID-19 pandemic, yet it has played a crucial role with widespread utilization specifically during the pandemic, preventing the discontinuation of clinical care and helping to avoid patients' isolation [15,16].
During lockdown periods, authors from Asia, Europe, and the USA reported that the amount of remote and telematic healthcare provided to RMDs patients increased from 50 to 150% [8,[17][18][19][20].According to Metha et al., 98% of rheumatologists modified their clinical practices during the pandemic, and 82% of them continue to use TM to guarantee continuity of care [21].During that time, telemedicine also prevented contact between healthcare professionals (HCPs) and patients, which decreased the risk of cross-infection in hospitals and contagion anxiety among the population [22].
At the best of our knowledge, with an exponential increase in published research on telemedicine, there is a great study heterogeneity in terms of methods and digital tools used, making implications and recommendations for clinical practice challenging to produce.Thus, the aim of the present scoping review is to identify the state of the art of the role of telemedicine in the field of rheumatology, with reference to the device used and PROs included by the authors.

Materials and methods
This scoping review was conducted following approaches outlined by Arksey and O'Malley and Levac et al., which provide a clear and rapid overview of relevant information regarding the telemedicine's role in rheumatology [22,23].The scoping review protocol was developed following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses Extension for Scoping Reviews (PRISMA-ScR) [24].Ethical approval was not sought as the data were publicly available.
The team conducted this scoping review through the following phases: (I) identifying the research question; (II) identifying relevant studies; (III) selecting the studies; (IV) extracting and charting the data; and (V) collating, summarizing, and reporting the results.

Phase I: identifying the research question
A research question was identified in accordance with the PEOS (Population, Exposure, Outcomes, and Study type) methodology (Table 1).RMDs patients were selected due to the prevalence of the disease and its high rate of complications.The exposure chosen included telemedicine, telenursing, and telehealth intervention, due to their growing utilization in healthcare and their widespread availability to persons nowadays.Telemedicine was defined as the utilization of remote communication technology aimed at treating, advising, screening, or reminding patients about their monitoring and treatment (e.g., text messaging, telephone communication, videoconferencing, and the use of mobile applications).Indeed, TM, in the present scoping review, refers to the provision of healthcare in situations where the health professional and the patient are not in the same physical location; then communication between the patients and the health professionals is activated, and data and information are moved.
The team hypothesized that employing telemedicine could enhance self-care among patients with RMDs and result in better economic, clinical, and humanistic outcomes (ECHO) [25].
Patients with RMDs were defined as adults (≥ 18 years old) affected by RMDs according to the American College of Rheumatology/European Alliance of Associations of Rheumatology (ACR/EULAR).
As for the outcome, disease activity and ECHO were included in the review.Disease activity likely refers to the level of severity or progression of the RMDs being studied.As for ECHO, it stands for economic, clinical, and humanistic outcomes, indicating a comprehensive approach to assessing the impact of the intervention.Economic outcomes were not searched within the field of the present scoping review, yet identified if present in the included papers, based on the definitions of direct and indirect cost, as measures of medical resource utilization such as hospitalization and medication costs, or evaluations of reduced productivity and lost working days.Clinical outcomes were defined as medical events that occurred because of healthcare services, including rate of readmission, emergency department visit, mortality, adverse drug events (including all-cause and serious adverse events), blood, and physiological parameters.Humanistic outcomes were defined as the consequences of disease or treatment on patient functional status or quality of life, including physical and social function, general health and well-being, and life satisfaction, including the relationship with the healthcare system (i.e., satisfaction with care, quality care).

Phase II: identifying relevant studies
In order to answer our research question and to identify the existing literature, the electronic databases MEDLINE, CINAHL (Cumulative Index to Nursing and Allied Health Literature), EMBASE (Excerpta Medica Database), APA (American Psychological Association), PsycINFO, and SCOPUS were investigated, using the following keywords to develop the search strategy for this scoping review: "rheumatic diseases," "telemedicine," "telenursing," "telehealth," and "patient reported outcome."The search strategy process was supervised and guided by the biomedical librarian.
After reading several documents, a search strategy combining Medical Subject Headings (MeSH) terms and free words was developed (Table 2).The different strings were divided into 3 dimensions: that relating to rheumatic diseases, that relating to telemedicine, and that relating to various patient outcomes.Each of these dimensions is joined by the Boolean operator "AND."Keywords were chosen either as the terms of the Thesaurus (MeSH for MEDLINE, Headings for CINAHL, and EMTree for EMBASE) or as common words combined through the Boolean operators "OR," "AND," and "NOT."Published records between January 1, 1990, and December 6, 2022, were downloaded for the screening process.
Records published between January 1, 1990, and December 6, 2022, were included for screening, limited to English language articles due to resource constraints.Exclusions comprised letters, editorials, news articles, reviews, theses, and other non-original qualitative or quantitative research papers.Additionally, studies lacking information about patients with RMDs or where telemedicine intervention was not explicitly detailed were excluded.Studies involving physicians, advanced practice nurses, physician associates, registered nurses, or pharmacists as presenters were included in the search.

Phase III: selecting the studies
The team that conducted the scoping review consisted of three nurses (KEA, MT, MRM) and a supervisor for the screening part with the role of resolving disagreements between the three reviewers (SB).The team members discussed and agreed on the relevance of the research question, examined whether the study design was appropriate, elaborated, and agreed on the research strategy.Retrieved literature was managed using EndNote™ (software version X9.3.3), which allowed the identification of duplicates to delete, according to literature title.After, titles and abstracts were screened independently by two reviewers (KEA and MRM, with the support of MT) according to the eligibility criteria.Then, retrieval of the full text was independently assessed in accordance with the inclusion/exclusion criteria.In case of disagreement, another reviewer (SB) was consulted.The study selection process is reported in the PRISMA flowchart.

Phase IV: extracting and charting the data
Data from each eligible article were extracted and compiled using a standardized excel sheet that was designed to guide the extraction of information from the records in accordance with the aim of the study, consisting of the following: authors, year and country of publication, study aim, study design, population and setting, intervention, results, outcomes measured, and tools used to measure them (Tables 3  and 4).Moreover, outcomes present in the included studies were divided following the ECHO model for the clinical, economic, and humanistic outcomes [25,26].Any kind of (hospital and primary care)

Phase V: collating, summarizing, and reporting the results
Findings concerning telemedicine interventions for patients with rheumatic diseases were then summarized and structured to offer a comprehensive overview of the existing evidence in this domain.Additionally, an appraisal of the quality of the evidence for the included studies was conducted by applying the Critical Appraisal Skills Programme (CASP) tools for both quantitative and qualitative studies [27].The total number of "yes" responses determined the quality of the studies, based on the established weight of the items.A CASP score > 7 indicates a study of good quality; a CASP score < 3 indicates a study of low quality.Each reviewer conducted an independent quality assessment.The CASP classifies the quality of evidence as three levels: high, moderate, and low.As stated for the screening process, disagreement on the assessment was solved by a third reviewer (SB).As for the mixed method study design, the mixed methods appraisal tool (MMAT) scale was used for quality assessment, as the CASP for mixed method studies is not available [28].We do not present an overall score for mixed method studies, as the authors of the MMAT scale discourage to do so, instead, they advise to provide a more detailed presentation of the ratings of each criterion to better inform the quality of the included studies.

Results
Using our search strategy, a total of 1507 records were identified, and then using Endnote software, 1078 were identified as duplicates and removed (Fig. 1).Of these, 1000 were excluded based on title or abstract.For the 78 studies remaining, 16 were excluded following a full-text review for failing to meet other inclusion criteria.Thus, a total of 62 studies were ultimately included in this scoping review: 54 quantitative study designs, 4 qualitative study designs, and 4 mixed method study design.Tables 3 and 4 summarize the data extracted from the 62 included studies.

Country of the selected studies
As for the country of publication of the included studies, 50% (31/62) was conducted in Europe; 14.5% (9/62) in United States of America and the same percentage in Asia; 12.9% (8/62) was conducted in New Zealand and Australia, while the remaining studies were conducted in South America or Saudi Arabia (Fig. 2).
e-Health tools used in the selected studies  5 and Fig. 4).

Humanistic outcomes reported in the selected studies
As for the humanistic outcomes according to the ECHO framework, the authors agreed on including a comprehensive categorization of the outcomes classifying them as regarding  5 and Fig. 4).Mental function included outcome measures as depression and anxiety, mental disability and psychological distress, catastrophizing, illness intrusiveness, fear of movement, and resilience.Physical function included outcome measures as the level of physical activity and mobility (i.e., general, hand, or upper limb function), health status and physical function, disease impact (i.e., RA and FMS), disability or physical distress, functional impact of the diseases (i.e., FMS), physical activity, and other outcomes such as fatigue, sleep quality, and stiffness.Health management included outcome measures such as disease knowledge (i.e., RA or FMS), treatment adherence, patient empowerment, activation and engagement, self-efficacy, self-care, self-management, coping strategies, health literacy, and patient education.One study included self-assessment and monitoring of Digital Ulcers (Dus) in SSc.
Health perception included outcome measures such as quality of life, illness perception, patient's satisfaction, beliefs about medical treatment, appropriateness of care received, and trust in the rheumatologist.Lastly, other outcome measures related to the experience of the patients with the e-health tool used are included in some studies: In particular, they assessed usefulness, suitability, user satisfaction, patients' and HCPs' experience, quality of the app, and user acceptance.

Quality assessment of the studies
We conducted the quality assessment of the included studies, as specified in the method section (Tables S6 to S12, Supplementary information).The three qualitative studies assessed were all of good quality.As for the quantitative studies, we assessed 15 observational/cohort studies with 5 good quality studies, 6 medium quality studies, and 4 low quality studies and 39 experimental/clinical trial with 14 good quality studies, 22 medium quality studies, and 3 low quality studies.

Discussion
The present scoping review aimed at identifying the state of the art regarding the application of digital technology, in terms of devices used and in terms of PROs assessed in the rheumatology field.The outcomes of this scoping review helped us to address our original query and comprehend the current state of the art when it comes to using all telemedicine tools in the context of rheumatology, and particularly those that are linked to specific outcomes.To date, it is commonly recognized that information technology is important and should be integrated to traditional care and assistance models.
Regarding the studies included in this scoping review, the methodological variety of study designs is undoubtedly something worth reflecting on.The presence of quantitative, qualitative, and mixed method studies highlights the heterogeneity of the aspects that can be investigated in the field of telemedicine in rheumatology.
Moreover, most of the studies included in this scoping review were conducted in European countries, USA, and also Australia make our results generalizable to RMDs populations, clearly after tailoring them to the clinical context.
As for the disease prevalence, OA and RA are the most studied diseases among RMDs, followed by CTDs and FMS.Indeed, several studies addressed telemedicine for patients with RA and OA suggesting that healthcare facilities are more likely to invest in telemedicine for these conditions due to their high prevalence.
Regarding the outcomes assessed for RA, quality of life and disease activity are present in the majority of the studies [29][30][31][32][33][34][35][36].In line with the literature, these are important outcomes to assess when dealing with arthritis as the disease affects the quality of life of RA patients by 93% and the ability to perform even the simplest daily gestures, such as opening a bottle, performing activities of daily life, climbing stairs, dressing, or washing by 85% [37].
In addition, disease activity is a pivotal parameter to include and monitor over time for all arthritis patients; in fact, specific questionnaires, that integrate inflammatory indices such as CRP or ESR in addition to patient-reported issues and health status, are widely used among the scientific and clinical community [38].
In the present review, the heterogeneity emerged from the included studies in terms of disease diagnoses, outcomes evaluated, and digital technologies used.Indeed, multiple tools of telemedicine, namely more than 1/3 of them used websites and online platforms, almost 1/3 used mobile applications, and the last 1/3 used telephone contacts.Video-consultations and wearable devices are less likely to be used when assessing or treating patients with RMDs.Furthermore, most studies that compare e-health interventions with standard of care, or standard visits and assistance, found that the use of telemedicine produces either positive or non-inferior outcomes [42,46,47,[49][50][51].These studies highlight the significance of both the patient's adherence to the treatment plan and the monitoring provided by HCPs, regardless of the digital tool utilized, which represent fundamental outcomes for chronic inflammatory RMDs.Moreover, these factors are crucial for nursing practice, indeed, according to Gordon's functional health patterns of health perception/management and activity/exercise, nurses, and HCPs in general, can plan interventions to promote treatment adherence while maintaining an appropriate quantity of physical activity [52].
When it comes to chronicity, self-management, and patient's autonomy in managing their illness are generally encouraged by specific mobile applications, and nearly all findings indicate that the use of these e-tools has a positive effect on PROMs.Indeed, also other studies are addressing the need to define the role of digital tools regarding selfmanagement in RMDs patients [53].
Essentially, these tools allow the patients to self-manage and monitor their condition, with direct access to relevant information and data about their disease, while encouraging patient's autonomy.
When telephone contacts are used, findings from the review showed that patient outcomes are favorable specifically regarding disease activity and disease management.Indeed, compared to websites and smartphone apps, telephone contact is more likely to offer inclusivity for patients with lower digital health literacy.This aspect and related factors have not received enough research attention, yet technology resources are today widely available for patients, thus evaluating that the patients' digital health literacy level is mandatory and determine its casual factors [54].
To summarize, the comprehensive disease monitoring, consistent with the person-centered approach, is in accordance with the results of the review, especially when we look at the humanistic outcomes.In fact, dealing with mental and physical function, health management, and perceptions allow us to assure a comprehensive assessment required for RMDs.
In addition, it is critical to recognize and detect timely significant symptoms or flare-ups to refer patients to face-toface consultations and prevent further complications.
Indeed, before implementing digital health interventions, a thorough evaluation of the patient in terms of clinical condition, health literacy level, and social context of life is required to provide international working groups' protocols, recommendations, and guidelines.

Strengths and limitations
This review has some strengths.First, diverse study designs provide a holistic understanding of telemedicine interventions.Second, valuable insights into improved disease management and patient-reported outcomes are highlighted.However, the clinical heterogeneity of tele-healthcare interventions and methodological bias can also constrain the evidence for effectiveness, but they support the personalization of clinical care paths.

Implications for clinical practice and research
The review underscores the need to critically evaluate research quality and tailor care pathways in telemedicine for rheumatological conditions.While digital tools hold promise, challenges such as technological diversity and patient health literacy require attention.
Future studies should focus on integrating patient perspectives for effective implementation and person-centered care.
In conclusion, the present scoping review showed the heterogeneity of digital tools in the field of rheumatology, which underlines the difficulty in transferring to clinical practice the results of valid studies.Clearly, OA and RA are the most studied diseases among RMDs, and effective tele-rehabilitation models have been presented, along with the application of the tight control strategy that recently emerged to be necessary in disease monitoring to achieve disease remission.Future studies should focus on summarizing and producing clinical recommendations for RMDs and developing studies on other RMDs, such as CTDs or FMS.

Fig. 3
Fig. 3 Prevalence of e-health tools used in the included studies, shown per type of study design

Fig. 4
Fig. 4 Outcome measures reported following the ECHOs classification

Table 3
Data extraction for quantitative studies